Background: Dermoid cysts are benign neoplasms deriving from ectoderm and mesoderm and can be found in a variety of locations. Uterine leiomyomas are benign tumors that grow within the muscle tissue of the uterus. Only a few cases have been reported and cited in the literature on dermoid cysts of the urinary bladder, and can be confused with urogenital masses. This paper reports the first case of a dermoid cyst deriving from the bladder dome presenting as subserosal uterine leiomyoma. Case: A 44-year-old woman was admitted with a history of chronic pelvic pain. After gynecological examination and ultrasonography a subserous uterine myoma was suspected. Tumor resection was performed and the histopathological diagnosis was a dermoid cyst. Conclusion: To our knowledge this is the first case report of a woman with a dermoid cyst presenting as a subserous uterine myoma.